Khouloud Marzouk, Nader Slama, Ahlem Bellalah, Ines Safra, Montacer Hafsi, Rayhan Chabbeh
Department of obstetrics and gynecology, Tahar Sfar university hospital 5111 Mahdia, Tunisia.
Departmentof hematology, Fattouma Bourguiba university hospital Monastir, Tunisia.
Leuk Res Rep. 2024 May 21;21:100462. doi: 10.1016/j.lrr.2024.100462. eCollection 2024.
Blastic plasmacytoid dendritic cell neoplasm (BPDCN) is a rare hematological malignancy characterized by the proliferation of plasmacytoid dendritic cells with a blast-like appearance. It usually presents in elderly people, and clinical manifestations include nodular blue-violet skin lesions, bone marrow infiltration and, less frequently, extramedullary involvement. Gynecological manifestation (breast mass and exocervical lesion) is an unusual and rare presentation. Herein, we report the case of a 51-year-old woman patient who presented with a history of a rapidly growing and bleeding breast mass, along with a decline in general health. Notably, the disease had multifocal involvement, affecting the breast, uterine cervix, and cervical lymphadenopathy. Biopsies were performed on the breast mass and cervical lesion. Histopathological examination showed a diffuse lymphoid proliferation. The neoplastic cells show immunoreactivity for CD45 and CD56. The myelogram showed a 50 % excess of blasts with a heterogeneous appearance with the presence of cells that could suggest dendritic plasmacytoid cells. Bone marrow immunophenotyping showed the presence of blast-like cells that were positive for CD4, CD56, CD123, which supported the diagnosis of BPDCN. Despite initiating chemotherapy, the patient's condition rapidly deteriorated, highlighting the aggressive nature of BDCP. This case underscores the importance of early detection and the need for further research to improve outcomes for this rare condition.
母细胞样浆细胞样树突状细胞肿瘤(BPDCN)是一种罕见的血液系统恶性肿瘤,其特征是具有原始样外观的浆细胞样树突状细胞增殖。它通常发生于老年人,临床表现包括结节性蓝紫色皮肤病变、骨髓浸润,较少见的是髓外受累。妇科表现(乳腺肿块和宫颈外病变)是一种不寻常且罕见的表现。在此,我们报告一例51岁女性患者,她有快速生长且出血的乳腺肿块病史,同时全身健康状况下降。值得注意的是,该疾病有多灶性受累,累及乳腺、子宫颈和颈部淋巴结病。对乳腺肿块和宫颈病变进行了活检。组织病理学检查显示弥漫性淋巴细胞增殖。肿瘤细胞对CD45和CD56呈免疫反应性。骨髓检查显示原始细胞增多50%,外观异质性,存在提示树突状浆细胞样细胞的细胞。骨髓免疫表型分析显示存在对CD4、CD56、CD123呈阳性的原始样细胞,支持BPDCN的诊断。尽管开始了化疗,但患者的病情迅速恶化,突出了BDCP的侵袭性。该病例强调了早期检测的重要性以及进一步研究以改善这种罕见疾病治疗结果的必要性。